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MHC class I deficiency 2(MHC1D2)

MedGen UID:
1862135
Concept ID:
C5935617
Disease or Syndrome
Synonyms: MHC CLASS I DEFICIENCY 2; MHC1D2
 
Gene (location): TAP2 (6p21.32)
 
Monarch Initiative: MONDO:0971011
OMIM®: 620813

Definition

MHC class I deficiency-2 (MHC1D2) is an autosomal recessive immunologic disorder characterized by the onset of chronic bacterial sinobronchial infections in the first or second decades of life. Nasal polyps are common, and chronic necrotizing granulomatous lesions affecting the nasal cavity, upper respiratory tract, or skin, which can be severe, may develop later. The disorder is progressive and patients may develop bronchiectasis and respiratory failure. Patient cells have decreased or absent expression of MHC type I (HLA class I) antigens on the cell surface (summary by de La Salle et al., 1994; Moins-Teisserenc et al., 1999). For a discussion of genetic heterogeneity of MHC class I deficiency, see MHC1D1 (604571). [from OMIM]

Clinical features

From HPO
Recurrent infection of the gastrointestinal tract
MedGen UID:
343135
Concept ID:
C1854495
Disease or Syndrome
Recurrent infection of the gastrointestinal tract.
Bronchiectasis
MedGen UID:
14234
Concept ID:
C0006267
Disease or Syndrome
Persistent abnormal dilatation of the bronchi owing to localized and irreversible destruction and widening of the large airways.
Chronic bronchitis
MedGen UID:
3084
Concept ID:
C0008677
Disease or Syndrome
Chronic inflammation of the bronchi.
Recurrent respiratory infections
MedGen UID:
812812
Concept ID:
C3806482
Finding
An increased susceptibility to respiratory infections as manifested by a history of recurrent respiratory infections.
Chronic sinusitis
MedGen UID:
101751
Concept ID:
C0149516
Disease or Syndrome
A chronic form of sinusitis.
Recurrent viral infections
MedGen UID:
332357
Concept ID:
C1837066
Finding
Increased susceptibility to viral infections, as manifested by recurrent episodes of viral infection.
Severe viral infection
MedGen UID:
479058
Concept ID:
C3277428
Finding
An unusually severe viral infection.
Cytoplasmic antineutrophil antibody positivity
MedGen UID:
1690999
Concept ID:
C5139210
Finding
The presence of autoantibodies in the serum that react against proteins predominantly expressed in cytoplasmic granules of neutrophils.
Chronic rhinitis
MedGen UID:
3086
Concept ID:
C0008711
Disease or Syndrome
Chronic inflammation of the nasal mucosa.
Nasal polyposis
MedGen UID:
6524
Concept ID:
C0027430
Anatomical Abnormality
Polypoidal masses arising mainly from the mucous membranes of the nose and paranasal sinuses. They are freely movable and nontender overgrowths of the mucosa that frequently accompany allergic rhinitis.

Term Hierarchy

CClinical test,  RResearch test,  OOMIM,  GGeneReviews,  VClinVar  

Recent clinical studies

Etiology

Calligaris M, Spanò DP, Bonelli S, Müller SA, Carcione C, D'apolito D, Amico G, Miele M, Di Bella M, Zito G, Nuti E, Rossello A, Blobel CP, Lichtenthaler SF, Scilabra SD
Cell Mol Life Sci 2024 Apr 4;81(1):163. doi: 10.1007/s00018-024-05201-7. PMID: 38570362Free PMC Article
Garrido F, Romero I, Aptsiauri N, Garcia-Lora AM
Int J Cancer 2016 Jan 15;138(2):271-80. Epub 2014 Dec 13 doi: 10.1002/ijc.29375. PMID: 25471439
Yoshida R
Adv Immunol 2014;124:207-47. doi: 10.1016/B978-0-12-800147-9.00007-8. PMID: 25175777
Hanna S, Etzioni A
J Allergy Clin Immunol 2014 Aug;134(2):269-75. Epub 2014 Jul 4 doi: 10.1016/j.jaci.2014.06.001. PMID: 25001848
Yong K, Khwaja A
Blood Rev 1990 Dec;4(4):211-25. doi: 10.1016/0268-960x(90)90001-9. PMID: 1706206

Diagnosis

Lau VWC, Mead GJ, Varyova Z, Mazet JM, Krishnan A, Roberts EW, Prota G, Gileadi U, Midwood KS, Cerundolo V, Gérard A
Nat Commun 2025 Jan 2;16(1):2. doi: 10.1038/s41467-024-54791-0. PMID: 39746898Free PMC Article
Torrejon DY, Galvez M, Abril-Rodriguez G, Campbell KM, Medina E, Vega-Crespo A, Kalbasi A, Comin-Anduix B, Ribas A
Cancer Immunol Res 2023 Dec 1;11(12):1642-1655. doi: 10.1158/2326-6066.CIR-23-0139. PMID: 37801341Free PMC Article
Hanna S, Etzioni A
J Allergy Clin Immunol 2014 Aug;134(2):269-75. Epub 2014 Jul 4 doi: 10.1016/j.jaci.2014.06.001. PMID: 25001848
Raulet DH
Adv Immunol 1994;55:381-421. doi: 10.1016/s0065-2776(08)60514-3. PMID: 8304235
Kärre K
Semin Immunol 1993 Apr;5(2):127-45. doi: 10.1006/smim.1993.1016. PMID: 8504217

Therapy

Eguren-Santamaria I, Fernández de Piérola E, Camps G, Martín-Muñoz P, Campos M, Cuculescu D, Aguilera-Buenosvinos I, Rodríguez López I, Salido-Vallejo R, Alexandru R, De Andrea CE, Álvarez-Gigli L, Berraondo P, Melero I, Sanmamed MF
J Immunother Cancer 2024 Sep 6;12(9) doi: 10.1136/jitc-2023-008516. PMID: 39244214Free PMC Article
de Vries NL, van de Haar J, Veninga V, Chalabi M, Ijsselsteijn ME, van der Ploeg M, van den Bulk J, Ruano D, van den Berg JG, Haanen JB, Zeverijn LJ, Geurts BS, de Wit GF, Battaglia TW, Gelderblom H, Verheul HMW, Schumacher TN, Wessels LFA, Koning F, de Miranda NFCC, Voest EE
Nature 2023 Jan;613(7945):743-750. Epub 2023 Jan 11 doi: 10.1038/s41586-022-05593-1. PMID: 36631610Free PMC Article
Vigneron N, Ferrari V, Van den Eynde BJ, Cresswell P, Leonhardt RM
J Immunol 2018 Oct 1;201(7):1875-1888. Epub 2018 Aug 22 doi: 10.4049/jimmunol.1701479. PMID: 30135181Free PMC Article
Thirdborough SM, Roddick JS, Radcliffe JN, Howarth M, Stevenson FK, Elliott T
Eur J Immunol 2008 Feb;38(2):364-9. doi: 10.1002/eji.200737832. PMID: 18196518
Rolli V, Radosavljevic M, Astier V, Macquin C, Castan-Laurell I, Visentin V, Guigné C, Carpéné C, Valet P, Gilfillan S, Bahram S
FEBS Lett 2007 Feb 6;581(3):394-400. Epub 2007 Jan 10 doi: 10.1016/j.febslet.2006.12.047. PMID: 17234189

Prognosis

Martín-Galiano AJ, Díez-Fuertes F, McConnell MJ, López D
Front Immunol 2021;12:732693. Epub 2021 Nov 25 doi: 10.3389/fimmu.2021.732693. PMID: 34899692Free PMC Article
Li L, Dong M, Wang XG
Chin Med J (Engl) 2016 Feb 20;129(4):448-55. doi: 10.4103/0366-6999.176084. PMID: 26879019Free PMC Article
de Jong RA, Boerma A, Boezen HM, Mourits MJ, Hollema H, Nijman HW
Int J Cancer 2012 Oct 15;131(8):1828-36. Epub 2012 Mar 9 doi: 10.1002/ijc.27449. PMID: 22287095
Garcia-Lora A, Algarra I, Garrido F
J Cell Physiol 2003 Jun;195(3):346-55. doi: 10.1002/jcp.10290. PMID: 12704644
Ruiz-Cabello F, Klein E, Garrido F
Immunol Lett 1991 Aug;29(3):181-9. doi: 10.1016/0165-2478(91)90168-a. PMID: 1769705

Clinical prediction guides

Lau VWC, Mead GJ, Varyova Z, Mazet JM, Krishnan A, Roberts EW, Prota G, Gileadi U, Midwood KS, Cerundolo V, Gérard A
Nat Commun 2025 Jan 2;16(1):2. doi: 10.1038/s41467-024-54791-0. PMID: 39746898Free PMC Article
Eguren-Santamaria I, Fernández de Piérola E, Camps G, Martín-Muñoz P, Campos M, Cuculescu D, Aguilera-Buenosvinos I, Rodríguez López I, Salido-Vallejo R, Alexandru R, De Andrea CE, Álvarez-Gigli L, Berraondo P, Melero I, Sanmamed MF
J Immunother Cancer 2024 Sep 6;12(9) doi: 10.1136/jitc-2023-008516. PMID: 39244214Free PMC Article
de Vries NL, van de Haar J, Veninga V, Chalabi M, Ijsselsteijn ME, van der Ploeg M, van den Bulk J, Ruano D, van den Berg JG, Haanen JB, Zeverijn LJ, Geurts BS, de Wit GF, Battaglia TW, Gelderblom H, Verheul HMW, Schumacher TN, Wessels LFA, Koning F, de Miranda NFCC, Voest EE
Nature 2023 Jan;613(7945):743-750. Epub 2023 Jan 11 doi: 10.1038/s41586-022-05593-1. PMID: 36631610Free PMC Article
Brehm MA, Kenney LL, Wiles MV, Low BE, Tisch RM, Burzenski L, Mueller C, Greiner DL, Shultz LD
FASEB J 2019 Mar;33(3):3137-3151. Epub 2018 Nov 1 doi: 10.1096/fj.201800636R. PMID: 30383447Free PMC Article
Garrido F, Romero I, Aptsiauri N, Garcia-Lora AM
Int J Cancer 2016 Jan 15;138(2):271-80. Epub 2014 Dec 13 doi: 10.1002/ijc.29375. PMID: 25471439

Supplemental Content